Langerhans Cell Histiocytosis with Atypical Intervertebral Disc and Sacroiliac Joint Involvement Mimicking Osteoarticular Tuberculosis in an Adult

Volume: 33 Number: 5 September 1, 2016
  • Zeynep Maraş Özdemir
  • Ayşegül Sağır Kahraman
  • Cemile Ayşe Görmeli
  • Reşit Sevimli
  • Nusret Akpolat
EN

Langerhans Cell Histiocytosis with Atypical Intervertebral Disc and Sacroiliac Joint Involvement Mimicking Osteoarticular Tuberculosis in an Adult

Abstract

Background: Langerhans cell histiocytosis (LCH), typically found in children, is a rare single or multisystem disorder with a wide range of clinical and radiological manifestations. Unusual presentations of LCH are occasionally encountered and it may be difficult to distinguish LCH from an infection or a benign or malignant tumor. Results: A 35-year-old female presented with pain in her back and left buttock, malaise, and weight loss, with a duration of several months. Her laboratory test results were within the normal ranges except for the levels of acute phase reactants, which were elevated. Magnetic resonance imaging and computed tomography revealed a unilateral destructive sacroiliac lesion, and multiple vertebral lesions with adjacent discal involvement and extensive soft tissue extensions. She was initially misdiagnosed with multifocal osteoarticular tuberculosis. An open biopsy and joint curettage was performed. Histopathological examination showed that she had LCH. Conclusion: To the best of our knowledge, this is the first case of LCH associated with a destructive unilateral sacroiliac lesion, discal involvement, and involvement of the adjacent vertebrae, in an adult patient; the LCH mimicked osteoarticular tuberculosis. Disease onset in adulthood is rare, and this can potentially delay diagnosis. Familiarity with the imaging features of unusual LCH manifestations is necessary to ensure accurate diagnosis and appropriate treatment.

Keywords

References

  1. 1. Stull MA, Kransdorf MJ, Devaney KO. Langerhans cell histiocytosis of bone. Radiographics 1992;12:801-23. [CrossRef]
  2. 2. Schmidt S, Eich G, Geoffray A, Hanquinet S, Waibel P, Wolf R, et al. Extraosseous langerhans cell histiocytosis in children. Radiographics 2008;28:707-26. [CrossRef]
  3. 3. Arico M, Girschikofsky M, Genereau T, Klersy C, McClain K, Grois N, et al. Langerhans cell histiocytosis in adults. Report from the International Registry of the Histiocyte Society. Eur J Cancer 2003;39:2341–8. [CrossRef]
  4. 4. Amini B, Kumar R, Wang WL. Soft tissue Langerhans cell histiocytosis with secondary bone involvement in extremities: evolution of lesions in two patients. Skeletal Radiol 2013;42:1301-9. [CrossRef]
  5. 5. Samet J, Weinstein J, Fayad LM. MRI and clinical features of Langerhans cell histiocytosis (LCH) in the pelvis and extremities: can LCH really look like anything? Skeletal Radiol 2016;45:607-13. [CrossRef]
  6. 6. Onal C, Oymak E, Reyhan M, Canpolat T, Ozyilkan O. Multifocal soft tissue Langerhans’ cell histiocytosis treated with PETCT based conformal radiotherapy. Jpn J Radiol 2015;33:603-6. [CrossRef]
  7. 7. Mondal D, Julka PK, Jana M, Walia R, Chaudhuri T. Langerhans cell histiocytosis of atlantoaxial joint in a middle-aged man presenting with deafness as first symptom and soft-tissue mass at neck showing excellent response to radiotherapy alone: Report of an extremely rare and unusual clinical condition and review of literature. Ann Indian Acad Neurol 2014;17:429-32. [CrossRef]
  8. 8. Hindman BW, Thomas RD, Young LW, Yu L. Langerhans cell histiocytosis: unusual skeletal manifestations observed in thirtyfour cases. Skeletal Radiol 1998;27:177-81. [CrossRef]

Details

Primary Language

English

Subjects

-

Journal Section

-

Authors

Zeynep Maraş Özdemir This is me

Ayşegül Sağır Kahraman This is me

Cemile Ayşe Görmeli This is me

Reşit Sevimli This is me

Nusret Akpolat This is me

Publication Date

September 1, 2016

Submission Date

September 1, 2016

Acceptance Date

-

Published in Issue

Year 2016 Volume: 33 Number: 5

APA
Özdemir, Z. M., Kahraman, A. S., Görmeli, C. A., Sevimli, R., & Akpolat, N. (2016). Langerhans Cell Histiocytosis with Atypical Intervertebral Disc and Sacroiliac Joint Involvement Mimicking Osteoarticular Tuberculosis in an Adult. Balkan Medical Journal, 33(5), 573-577. https://izlik.org/JA59HU74YR
AMA
1.Özdemir Z M, Kahraman AS, Görmeli CA, Sevimli R, Akpolat N. Langerhans Cell Histiocytosis with Atypical Intervertebral Disc and Sacroiliac Joint Involvement Mimicking Osteoarticular Tuberculosis in an Adult. Balkan Medical Journal. 2016;33(5):573-577. https://izlik.org/JA59HU74YR
Chicago
Özdemir, Zeynep Maraş, Ayşegül Sağır Kahraman, Cemile Ayşe Görmeli, Reşit Sevimli, and Nusret Akpolat. 2016. “Langerhans Cell Histiocytosis With Atypical Intervertebral Disc and Sacroiliac Joint Involvement Mimicking Osteoarticular Tuberculosis in an Adult”. Balkan Medical Journal 33 (5): 573-77. https://izlik.org/JA59HU74YR.
EndNote
Özdemir Z M, Kahraman AS, Görmeli CA, Sevimli R, Akpolat N (September 1, 2016) Langerhans Cell Histiocytosis with Atypical Intervertebral Disc and Sacroiliac Joint Involvement Mimicking Osteoarticular Tuberculosis in an Adult. Balkan Medical Journal 33 5 573–577.
IEEE
[1]Z. M. Özdemir, A. S. Kahraman, C. A. Görmeli, R. Sevimli, and N. Akpolat, “Langerhans Cell Histiocytosis with Atypical Intervertebral Disc and Sacroiliac Joint Involvement Mimicking Osteoarticular Tuberculosis in an Adult”, Balkan Medical Journal, vol. 33, no. 5, pp. 573–577, Sept. 2016, [Online]. Available: https://izlik.org/JA59HU74YR
ISNAD
Özdemir, Zeynep Maraş - Kahraman, Ayşegül Sağır - Görmeli, Cemile Ayşe - Sevimli, Reşit - Akpolat, Nusret. “Langerhans Cell Histiocytosis With Atypical Intervertebral Disc and Sacroiliac Joint Involvement Mimicking Osteoarticular Tuberculosis in an Adult”. Balkan Medical Journal 33/5 (September 1, 2016): 573-577. https://izlik.org/JA59HU74YR.
JAMA
1.Özdemir Z M, Kahraman AS, Görmeli CA, Sevimli R, Akpolat N. Langerhans Cell Histiocytosis with Atypical Intervertebral Disc and Sacroiliac Joint Involvement Mimicking Osteoarticular Tuberculosis in an Adult. Balkan Medical Journal. 2016;33:573–577.
MLA
Özdemir, Zeynep Maraş, et al. “Langerhans Cell Histiocytosis With Atypical Intervertebral Disc and Sacroiliac Joint Involvement Mimicking Osteoarticular Tuberculosis in an Adult”. Balkan Medical Journal, vol. 33, no. 5, Sept. 2016, pp. 573-7, https://izlik.org/JA59HU74YR.
Vancouver
1.Zeynep Maraş Özdemir, Ayşegül Sağır Kahraman, Cemile Ayşe Görmeli, Reşit Sevimli, Nusret Akpolat. Langerhans Cell Histiocytosis with Atypical Intervertebral Disc and Sacroiliac Joint Involvement Mimicking Osteoarticular Tuberculosis in an Adult. Balkan Medical Journal [Internet]. 2016 Sep. 1;33(5):573-7. Available from: https://izlik.org/JA59HU74YR